Total submissions: 2
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Blueprint Genetics | RCV000788125 | SCV000927134 | pathogenic | not provided | 2017-01-26 | criteria provided, single submitter | clinical testing | |
Labcorp Genetics |
RCV001385304 | SCV001585113 | pathogenic | Hyper-IgM syndrome type 2 | 2022-06-26 | criteria provided, single submitter | clinical testing | This sequence change replaces methionine, which is neutral and non-polar, with threonine, which is neutral and polar, at codon 139 of the AICDA protein (p.Met139Thr). For these reasons, this variant has been classified as Pathogenic. Experimental studies have shown that this missense change affects AICDA function (PMID: 22715099). Algorithms developed to predict the effect of missense changes on protein structure and function (SIFT, PolyPhen-2, Align-GVGD) all suggest that this variant is likely to be disruptive. ClinVar contains an entry for this variant (Variation ID: 636339). This missense change has been observed in individual(s) with autosomal recessive hyper IgM syndrome (HIGM) (PMID: 27142677). It has also been observed to segregate with disease in related individuals. This variant is present in population databases (rs200858797, gnomAD 0.2%). |